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T699P

Category 3/4 — Most DruggableLikely pathogenicLumenal · predictedσ-1 candidateEditorial
ThreonineProline at position 699 · C-terminal lumenal domain (653-869) · WFS1 (Wolframin)

Threonine → Proline at position 699 in wolframin's C-terminal lumenal domain. ClinVar Likely pathogenic for Wolfram syndrome 1. AlphaMissense 0.914, DynaMut2 ΔΔG -0.14 kcal/mol (mild destabilising). Proline-introduction adjacent to W700 (Cat 2 outlier region).

Interactive 3D Structure

Wild-type reference
Wild-type T699 — hydrogen bond to F825
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DynaMut2 mutant · T699P
Mutant P699 — polar contact contact to F825 lost
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Bond changes · DynaMut2 interaction analysis

1 lost0 gained1 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondF825F825Preserved
Polar contactF825Lost

Lost / gained / preserved interatomic contacts at the variant residue, from the DynaMut2 (Arpeggio) interaction analysis of the wild-type and energy-minimized mutant structures.

Computational Predictions

DynaMut2 ΔΔG
-0.14kcal/mol
Destabilising — mild
AlphaMissense
0.914
LPath
AlphaFold pLDDT
89
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable

Clinical Evidence

ClinVar classificationLikely pathogenic
Review statuscriteria provided, single submitter
Associated conditionsWolfram syndrome 1
InheritanceWolfram syndrome 1 (AR) documented.
Population frequency (gnomAD v4)Ultra-rare · AF 0.000068%
cDNA changec.2095A>C
ClinVar accessionVCV003767960
Last evaluated2025/01/27 00:00

Observed at very low frequency in gnomAD.

Structural Context

Position 699 sits in wolframin's C-terminal lumenal domain, immediately preceding W700 (the position of W700S — Cat 2 outlier — and W700C in the Atlas). The AlphaFold model places T699 within 5 Å of TRP700 (2.4 Å), VAL698 (2.5 Å), PHE825 (3.4 Å — W700's aromatic-stacking partner), and SER826 (4.3 Å).

The wild-type threonine at 699 sits in immediate contact with W700 and the F825 aromatic neighbor. The hydroxyl group H-bonds locally and stabilizes the geometry that supports W700's aromatic packing with F825.

Replacing T699 with proline introduces a backbone kink immediately upstream of W700, perturbing the precise W700-F825 π-stacking geometry the Atlas's W700C card identifies as the key functional contact. The |ΔΔG| of 0.14 is small (fold absorbs), but the functional consequence — disrupted W700 stacking — is the same severe pathogenic mechanism that pulls W700C into pathogenic territory.

Amino-acid chemistry
Threonine (T) → Proline (P) — small polar hydroxyl-bearing residue replaced by rigid helix-breaking residue.
Position in the protein
C-terminal lumenal domain · position 699 in the ER lumen (pLDDT 89). Immediately adjacent to W700.

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.14 — fold survives. AlphaMissense 0.914 + Wolfram 1 confirm severe functional consequence.

Mechanism is backbone kink upstream of W700 that perturbs W700-F825 π-stacking geometry. Therapeutic strategy: same target as W700C/W700S — the W700-F825 aromatic stacking microregion.

Why this matters

T699P, W700C, W700S all converge on the W700-F825 π-stacking interface — three Atlas variants at one therapeutic target. The Atlas surfaces these as a coherent multi-variant rescue opportunity.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the T699P PDF below and upload it to Wolfram Intelligence to generate therapeutic-strategy proposals — guanidinium mimetics, sigma-1 agonist docking, NAC thiol-capping. NAC is already on the bench-testing list.

Download T699P PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Topological domain653869 · Lumenal
Natural variant699699 · in DFNA6; dbSNP:rs28937894